[Update on a diagnostic test for choroideremia: the protein truncation test (PTT)]

J Fr Ophtalmol. 1998 May;21(5):345-50.
[Article in French]

Abstract

Purpose: The aim of this study was to define the RT-PCR-PTT parameters for CHM gene analysis and to evaluate its interest as a method for CHM mutation screening.

Methods: The entire CHM coding region was reversed-transcribed in three overlapping cDNA segments (RT-PCR) which were amplified and further analyzed by PTT after in vitro transcription/translation.

Results: This strategy enabled us to detect a truncated peptide in each of the 6 unrelated patients from southern France who were investigated. The mutation was further characterized by direct sequencing of the RT-PCR product.

Conclusion: In CHM gene, all conditions are present to make the RT-PCR-PTT strategy the method of choice for mutation screening. As a result of the simplified protocol described in this study, the families of the patients could benefit from accurate carrier-status assessment.

MeSH terms

  • Adaptor Proteins, Signal Transducing
  • Alkyl and Aryl Transferases*
  • Carrier Proteins / analysis
  • Carrier Proteins / chemistry
  • Carrier Proteins / genetics*
  • Choroideremia / diagnosis*
  • Choroideremia / genetics
  • DNA Mutational Analysis
  • DNA, Complementary / genetics
  • Eye Proteins / analysis
  • Eye Proteins / chemistry
  • Eye Proteins / genetics*
  • Female
  • France
  • Genes, Recessive
  • Genetic Carrier Screening / methods*
  • Genetic Testing / methods*
  • Humans
  • Male
  • Protein Biosynthesis
  • Reverse Transcriptase Polymerase Chain Reaction*
  • Sequence Analysis, DNA
  • Sequence Deletion*
  • X Chromosome / genetics*
  • rab GTP-Binding Proteins*

Substances

  • Adaptor Proteins, Signal Transducing
  • CHM protein, human
  • Carrier Proteins
  • DNA, Complementary
  • Eye Proteins
  • Alkyl and Aryl Transferases
  • rab GTP-Binding Proteins